Manifestaciones bucales de la enfermedad celíaca y alergia al trigo: Reporte de tres casos y revisión literatura / Oral Manifestations of Celiac Disease and Wheat Allergy: Report of Three Cases and Review of Literature

Resumen: La Enfermedad Celíaca (EC) es una enfermedad sistémica inmunomediada, provocada por el gluten y prolaminas relacionadas, en individuos genéticamente susceptibles. Se caracteriza por manifestaciones clínicas dependientes del gluten, anticuerpos específicos de EC, haplotipos HLA DQ2 o DQ8 y enteropatía. La mayoría de los pacientes son asintomáticos o con manifestaciones leves. Afecta principalmente el sistema gastrointestinal causando síntomas y signos como diarrea, dolor abdominal recurrente y distensión abdominal; además manifestaciones extradigestivas. Los pacientes con EC pueden tener manifestaciones bucales que incluyen estomatitis aftosa recurrente (EAR), hipoplasia del esmalte, glositis atrófica, entre otras. El diagnóstico de EC se basa en la historia clínica, serología, endoscopía e histología. Existen otras entidades que deben diferenciarse de la EC, como la sensibilidad al gluten no celíaca y la alergia al trigo. El objetivo de este trabajo es presentar tres pacientes con antecedentes de EAR y otras manifestaciones clínicas asociadas al diagnóstico precoz de EC y alergia alimentaria.

Abastract: Celiac Disease (CD) is an immune-mediated systemic disease, caused by gluten and related prolamins, in genetically susceptible individuals. It is characterized by gluten-dependent clinical manifestations, CD-specific antibodies, HLA DQ2 or DQ8 haplotypes, and enteropathy. Most patients are asymptomatic or with mild manifestations. It mainly affects the gastrointestinal system causing symptoms and signs such as diarrhea, recurrent abdominal pain and abdominal distension; also extradigestive manifestations. Patients with CD may have oral manifestations that include recurrent aphthous stomatitis (RAS), enamel hypoplasia, atrophic glossitis, among others. The diagnosis of CD is based on clinical history, serology, endoscopy and histology. There are other entities that should be differentiated from CD, such as non-celiac gluten sensitivity and wheat allergy. The aim of this study is to present three patients with a history of RAS and other clinical manifestations associated with the early diagnosis of CD and food allergy.

Clinical characteristics of 21 children with wheat allergy / 中华实用儿科临床杂志

Objective:To analyze and summarize the clinical features of wheat allergy in children, and to increase the awareness and diagnosis of food allergy in children.Methods:Children suspected with wheat allergy in the Department of Allergy, Children′s Hospital of Capital Institute of Pediatrics from December 2018 to November 2019 were included.Clinical data were collected, including the demographic profile, serum allergen specific immunoglobulin E(IgE) detection, peripheral eosinophil detection, and skin prick testing.Results:A total of 21 cases were included, consisting of 15 males and 6 females, aged (21.5±12.8) months.The children developed allergic symptoms after intake of wheat products or contact with wheat flour.The most common clinical manifestations were cutaneous (20/21 cases, 95.2%), followed by respiratory (7/21 cases, 33.3%), digestive (6/21 cases, 28.6%) and nervous (1/21 cases, 4.8%) symptoms.Eleven children had anaphylaxis.Laboratory allergy test showed that the detection level of serum wheat-specific IgE in 21 children was 1.46 to 100.00 kU/L, and when the specific IgE level was in grade Ⅴ or above, the risk of serious allergic reaction was increased in children ( P<0.05). Five of them tested positive in the wheat skin prick test.The peripheral eosinophil ratio in children was 0.04-0.12.All children had a history of other food allergies and family allergies. Conclusions:Wheat allergy in children has early onset and diversified clinical manifestations, mainly skin symptoms.Serum specific IgE detection shows an important role in the diagnosis and evaluation of wheat allergy.The highly sensitized individuals are prone to anaphylaxis.

Enfermedad celíaca, sensibilidad no celíaca al gluten y alergia al trigo: comparación de patologías diferentes gatilladas por un mismo alimento / Celiac disease, non celiac gluten sensitivity and wheat allergy: comparison of 3 different diseases triggered by the same food

El gluten y otras proteínas relacionadas del trigo, centeno y cebada, tienen propiedades antigénicas que pueden desencadenar reacciones adversas en individuos susceptibles. La enfermedad celíaca fue la primera patología en que se estableció relación causal con estas proteínas alimentarias. Recientemente se han descrito la alergia al trigo y la sensibilidad no celíaca al gluten. Si bien sus formas de presentación clínica y su relación con la ingesta pueden ser similares, sus mecanismos patogénicos, forma de diagnóstico y tratamiento difieren. Dado que su prevalencia en conjunto es relativamente alta, resulta necesario que los médicos de atención primaria y pediatras se familiaricen con estas patologías, sepan cómo diferenciarlas y enfrentarlas. El objetivo de esta revisión es comparar los principales aspectos de epidemiología, fisiopatología, diagnóstico y tratamiento de estas 3 condiciones.

Gluten and other related proteins of the wheat, rye and barley, have antigenic properties that may trigger adverse reactions in susceptible individuals. Celiac disease was the first pathology with clear causal association related to the intake of these proteins. Recently, wheat allergy and non celiac gluten sensitivity have been described. Although, clinical presentation and its relation with protein ingestion may be similar and elicit confusion, their pathogenic mechanism, diagnosis and treatment are quite different. Since the prevalence of these diseases is relatively high as a whole, it is essential that these become familiar to primary care doctors and general pediatricians, thus they will know how to differentiate and face them. The aim of this review is to compare the main aspects of epidemiology, pathofisiology, diagnosis and treatment of these 3 conditions.

Characteristics of Anaphylaxis in 907 Chinese Patients Referred to a Tertiary Allergy Center: A Retrospective Study of 1,952 Episodes

PURPOSE: Comprehensive evaluation of anaphylaxis in China is currently lacking. In this study, we characterized the clinical profiles, anaphylactic triggers, and emergency treatment in pediatric and adult patients. METHODS: Outpatients diagnosed with "anaphylaxis" or "severe allergic reactions" in the Department of Allergy, Peking Union Medical College Hospital from January 1, 2000 to June 30, 2014 were analyzed retrospectively. RESULTS: A total of 1,952 episodes of anaphylaxis in 907 patients were analyzed (78% were adults and 22% were children). Foods are the most common cause (77%), followed by idiopathic etiologies (15%), medications (7%) and insects (0.6%). In food-induced anaphylaxis, 62% (13/21) of anaphylaxis in infants and young children (0-3 years of age) were triggered by milk, 59% (36/61) of anaphylaxis in children (4-9 years of age) were triggered by fruits/vegetables, while wheat was the cause of anaphylaxis in 20% (56/282) of teenagers (10-17 years of age) and 42% (429/1,016) in adults (18-50 years of age). Mugwort pollen sensitization was common in patients with anaphylaxis induced by spices, fruits/vegetables, legume/peanuts, and tree nuts/seeds, with the prevalence rates of 75%, 67%, 61%, and 51%, respectively. Thirty-six percent of drug-induced anaphylaxis was attributed to traditional Chinese Medicine. For patients receiving emergency care, only 25% of patients received epinephrine. CONCLUSIONS: The present study showed that anaphylaxis appeared to occur more often in adults than in infants and children, which were in contrast to those found in other countries. In particular, wheat allergens played a prominent role in triggering food-induced anaphylaxis, followed by fruits/vegetables. Traditional Chinese medicine was a cause of drug-induced anaphylaxis. Furthermore, exercise was the most common factor aggravating anaphylaxis. Education regarding the more aggressive use of epinephrine in the emergency setting is clearly needed.

Clinical and laboratory features, and quality of life assessment in wheat dependent exercise-induced anaphylaxis patients from central China / 华中科技大学学报（医学）（英德文版）

Wheat dependent exercise-induced anaphylaxis (WDEIA) is a rare but potentially severe food allergy caused by the combination of wheat ingestion and physical exercise. The impact of WDEIA on quality of life (QOL) is unclear. This study characterized the clinical and laboratory features and investigated the QOL in WDEIA patients from Central China. Twenty-eight WDEIA patients were analyzed, and QOL was measured by validated Chinese version Food Allergy Quality of Life Questionnaire-Adult Form (FAQLQ-AF) and Food Allergy Independent Measure (FAIM) after obtaining the diagnosis. The results showed that half of the patients were females. The median onset age was 37 years old. The symptoms occurred within 1 h after wheat ingestion (26/28). Symptoms of anaphylaxis included cutaneous (26/28), respiratory (11/28), gastro-intestinal (5/28) and cardiovascular manifestations (27/28). Skin prick tests were positive to salt soluble (89.3%) and salt insoluble wheat allergen extracts (100%). Positive rate to wheat, gluten and omega-5 gliadin specific IgE was 64.3%, 92.9% and 92.9% respectively. Specific IgE to omega-5 gliadin with a cut-off value 0.83 KU/L offered highly efficient diagnostic criterion for WDEIA (sensitivity: 89.3%; and specificity: 88.9%). The mean scores of FAQLQ-AF and FAIM were 4.70 and 4.98 respectively and level of anti-omega-5 gliadin IgE had positive correlations with FAQLQ scores. Thereby, WDEIA is commonly found in mid-age adults. In most cases, multi-organs especially skin and cardiovascular systems are involved. Salt insoluble wheat allergen skin test and serum specific IgE to gluten and omega-5 gliadin help to diagnose WDEIA. QOL in WDEIA patients is severely impaired.

Sensibilidad al gluten: presentación de tres casos / Gluten sensitivity: presentation of three cases

En los últimos años, ha cobrado mayor interés la existencia de un cuadro clínico muy similar al de la enfermedad celíaca, que no se ajusta a los cánones tradicionales de diagnóstico. Se trata de pacientes con una alta sospecha diagnóstica de enfermedad celíaca, que presentan serología y biopsia de intestino delgado normal. La literatura relata, desde la década del 80, la existencia de un síndrome que relaciona el gluten de la dieta con un efecto tóxico generador de síntomas gastrointestinales en presencia de una mucosa normal. A esta entidad se la denominó síndrome de Cooper-Cook. En los últimos años, ha habido numerosas publicaciones que hacen referencia a esta entidad, pero ahora bajo la denominación de sensibilidad al gluten. En el siguiente artículo, se presentan tres casos clínicos que hacen referencia a esta enfermedad.

In the last few years, the existence of a clinical profile similar to celiac disease has become important; this disease does not adapt to the traditional diagnosis canons. It is related to a number of patients who are diagnosed as having the celiac disease but present normal serology and small bowel's biopsy. Since the 80's, medical literature reports the existence of a syndrome that connects gluten diet with a toxic effect that produces gastrointestinal symptoms even though the mucosa remains normal. This disease is called the Cooper-Cook syndrome. Over the last few years, there have been lots of publications about this disease under the name "gluten sensitivity". In the following article, three clinical cases that refer to this condition are presented.

Successful wheat-specific oral immunotherapy in highly sensitive individuals with a novel multirush/maintenance regimen

We reported a successful oral immunotherapy (OIT) in 2 children with high wheat sensitivity (4 and 14 years old boys). Oral challenges indicated eliciting doses of 300 mg, and wheat flour of 30 mg. The OIT protocol includes 5 days of build-up phase in the hospital, intervening with 2 to 5 months of home maintenance phase. Patients could tolerate 45 g, and 60 g of wheat flour per day, respectively. We have demonstrated that OIT to a large amount of wheat in extremely sensitized patients could be achieved with a stepwise multi oral/maintenance regimen.

Wheat-Induced Anaphylaxis in Korean Adults: A Report of 6 Cases

Wheat is a common cause of food allergy. Wheat-induced anaphylaxis (WIA) and wheat-dependent exercise induced anaphylaxis (WDEIA) are severe forms of immunoglobulin E (IgE) mediated allergic reaction to wheat protein. As the diagnosis of WIA or WDEIA is not easy because of the risk of oral challenge, identification of specific IgE of various wheat proteins is helpful for diagnosis. In Korea, there are only a few reports on WIA in adults. We report six cases of WIA diagnosed on the basis of clinical history and specific IgE of wheat proteins or provocation test. For immunologic evaluation of severe wheat allergy including WIA and WDEIA, it is important to measure specific IgE to each component of wheat including gluten and omega-5 gliadin not just measuring wheat-specific IgE.

A Case of Gluten Allergy in a 4-Year-Old Boy With Recurrent Urticaria / 소아알레르기및호흡기학회지

Wheat is the most widely cultivated grain and an important source of food and dietary protein. Wheat proteins are classified based on extraction in different solvents, which are albumin, globulin, prolamin (gliadin) and glutenin. The term 'gluten' contains approximately equal amounts of gliadin and glutenin and is the major determinant of the properties of wheat flour conferring cohesiveness and viscoelasticity that allows its dough to be processed into many kinds of food. Gluten is known to be responsible for triggering celiac disease and wheat allergy. Wheat allergy is primarily an IgE-mediated response. Clinical manifestations of wheat allergy are similar to those of other food allergies, with symptoms on the skin, gut and respiratory tract. Recent studies have shown that IgE to gliadin can be an indicator for risk of severe immediate reaction-like anaphylaxis and wheat-dependent, exercise-induced anaphylaxis (WDEIA). However, current in vitro test reagents for the diagnosis of wheat allergy mainly contain water-soluble wheat protein and a small amount of gluten, so there are some limitations to diagnose gluten allergy. Furthermore, there is no acceptable method to measure gluten in food products for preparing effective gluten-free diet. To overcome these limitations and to improve quality of life of wheat allergy sufferers, more work is needed. We report a case of a 4-year-old boy with gluten allergy who presented with urticaria after ingestion kneaded wheat flour with a brief review of the literature.